Sudden breath-holding episodes during sleep in young children are potentially related to sudden infant death syndrome and other life-threatening events. Additionally, these episodes can negatively affect child’s growth and development.
Seo et al BMC Pediatrics (2017) 17:169 DOI 10.1186/s12887-017-0922-9 CASE REPORT Open Access My child cannot breathe while sleeping: a report of three cases and review Won Hee Seo1, Minkyu Park2, So-Hee Eun1, Seonkyeong Rhie3, Dae Jin Song1 and Kyu-Young Chae3* Abstract Background: Sudden breath-holding episodes during sleep in young children are potentially related to sudden infant death syndrome and other life-threatening events Additionally, these episodes can negatively affect child’s growth and development Case presentation: Here, we present cases of preschool children with similar paroxysmal nocturnal waking events associated with choking that had different etiologies (nocturnal frontal lobe epilepsy, nocturnal gastroesophageal reflux disease, and parasomnia, respectively) Conclusions: It is important to take into consideration the fact that breath spells during sleep can occur as a rare manifestation of parasomnia due to gastroesophageal reflux or as a symptom of nocturnal frontal lobe epilepsy Full video electroencephalography, polysomnography, and simultaneous gastric pH monitoring should be used for the differential diagnosis of sleep-related disorders, such as breath spells, in children Keywords: Breath-holding spell, Gastroesophageal reflux, Laryngospasm, Nocturnal seizure, Polysomnography, Sleep Background Sudden breath-holding and cyanotic events during sleep are known to cause health complications in young children These episodes can be frightening and cause intense hypnophobia Frequent nighttime awakening can also have detrimental effects on child’s growth and development, as well as adversely affect the health of the entire family The burden of caring for affected children can decrease family’s productivity, increase the incidence of family conflict-related stress, and even lead to maternal depression [1] Thus, the early diagnosis and management of nocturnal breath-holding episodes are critical for maintaining the quality of life of both the affected child and family A variety of conditions can cause nocturnal waking episodes in childhood Frequent nocturnal insomnias can be related to an acute medical condition, such as otitis media, sleep-onset association disorders caused by a lack of sleep training, such as childhood behavioral insomnia, primary sleep disorders, such as sleep-related breath * Correspondence: danielchae21@gmail.com Department of Pediatrics, CHA Bundang Medical Center, CHA University, 59 Yatapro, Seongnam, 13496 GyungGi Province, Korea Full list of author information is available at the end of the article disorder, or partial arousal parasomnias, such as night terrors and confusional arousals [2, 3] Paroxysmal nocturnal events are commonly misdiagnosed as epilepsy However, nocturnal epilepsy and parasomnias are distinct causes of breath-holding during sleep that should be differentiated [4, 5] In addition, laryngospasm is a rare cause of abrupt breath-holding during sleep that is caused by involuntary muscular contraction of the laryngeal cords [6, 7] Extraesophageal manifestations of the gastroesophageal reflux disease (GERD) are a known cause of laryngospasm Polysomnography (PSG) is the standard method for the diagnosis of sleep disorders Alternatively, video electroencephalography (EEG) can provide useful diagnostic information in individuals with a history of prominent motor activity during sleep [8, 9] However, the isolated use of these tools is often insufficient for the correct diagnosis of nocturnal events In this report, we present three cases of preschoolaged children who had similar symptoms (i.e., multiple abrupt waking with choking episodes every night), but received different diagnoses based on PSG with fullmontage of video-EEG, as well as 24-h esophageal pH monitoring © The Author(s) 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated Seo et al BMC Pediatrics (2017) 17:169 Page of Case presentation Case Case A 4-year-old girl was admitted complaining of 1–3 nightly episodes of sudden nocturnal waking with choking or coughing that had occurred over the previous 40 days The patient’s body weight (13 kg) and height (91 cm) were in the 3rd percentile for Korean girls according to the Korean growth charts The patient had no history of medical or neurological problems from birth, and no familial history of epilepsy or allergic disease The patient had sudden awakening events accompanied by intense fear due to difficulty breathing during sleep After each episode, the patient was unable to speak or cry, but these symptoms resolved in 1–3 with back percussion and reassurance from her parents Subsequently, the child would cry and return to sleep No abnormalities were detected by respiratory tests, neurological tests, full-montage EEG, or brain MRI Upon physical examination, the patient presented adenoid hypertrophy and it was noted that she had snoring episodes 1–2 times per week Her overnight PSG revealed repetitive choking and inspiratory stridor followed by sudden cortical arousal with obstructive apnea (Fig 2a) Simultaneous 24-h gastric pH monitoring revealed significant gastroesophageal reflux (GER) during nocturnal events (Fig 2b) Erosive esophagitis was subsequently found by gastroscopy The patient was diagnosed with nocturnal GERD and received anti-reflux therapy, including gastrokinetic agents and proton pump inhibitors (PPI) that completely resolved the symptoms within weeks A 4-year-old girl was admitted to our clinic with a years history of choking episodes during sleep that occurred 4–5 times almost every night The patient had no familial history of epilepsy or allergic disease, and her weight and height were within the normal range according to the Korean growth charts During the initial evaluation performed years before, brain magnetic resonance imaging (MRI) and standard PSG revealed no abnormalities Because the EEG revealed few focal epileptiform discharges in the left frontal area, the patient was prescribed an antiepileptic medication; however, her nocturnal spells persisted Upon the admission, we performed PSG with videoEEG Five choking episodes were observed on the video; the patient awakened suddenly, sat up, and experienced episodes of choking, air hunger, agitation, and intense fear; in two episodes, with the patient showed gagging gestures just after awakening During these periods, PSG recording and diagnostic parameters showed that the patient was in stage of non-rapid eye movement (NREM) sleep and experienced obstructive or mixed-type apnea, followed by arousal and awakening with dyspnea and tachycardia The peripheral oxygen saturation (Sp02) decreased to 83% During these episodes, the patient’s parents performed back percussion and provided reassurance Episodes typically resolved within 1–3 and the patient fell asleep again after each event We attempted a third diagnostic session of PSG with full-montage of video-EEG and simultaneous 24-h gastric pH monitoring in order to exclude the possibility of GERD Out of choking episodes, were associated with low-amplitude 13–14 Hz fast activities in the right or left frontal areas (Fz, F3, F4) that evolved into medium-to-high-amplitude bi-frontal sharp wave discharges intermixed with 6–7 Hz activities in the posterior cerebral region Accompanying obscuring muscle artifacts were observed and presented as sudden eye opening and right arm tonicity, followed by breathholding with or without agitation and gagging (Fig 1a, b) Several low pH (< 4) events were observed on simultaneous 24-h pH monitoring; however, these episodes did not coincide with the nocturnal breathholding spells The patient was diagnosed with frontal lobe epilepsy and experienced complete resolution of her nocturnal symptoms after intravenous administration of 20 mg/kg of sodium valproate, and then, the patient was prescribed oral valproate (Depakoke15mg/ kg/day, twice a day) month later, the patient had added topiramate On follow up until year after, it had been reported that the patient’s symptoms were well controlled with only sporadic manifestation of nocturnal episodes Case A 5-year-old girl was admitted with complaints of 2–3 nightly episodes of sudden waking and choking that occurred during the previous days The episodes usually occurred between 02:00–04:00 AM and were sometimes accompanied by coughing The patient had no familial history of epilepsy or allergic disease The patient’s parents witnessed sudden awakening accompanied by intense fear due to difficult breathing during sleep During some episodes, the patient complained of chest discomfort and dyspnea Each episode lasted 5– and resolved after back percussion and reassurance from the parents After each episode, the patient returned to sleep and was unable to recall the episode the following morning No abnormalities were detected by respiratory tests, neurological tests, full-montage EEG, or brain MRI Upon physical examination, the patient presented adenoid hypertrophy and it was noted that the patient had snoring episodes 3–4 times per week During the overnight PSG, the patientawoke3 times due to choking and crying during NREM and REM sleep for periods of Seo et al BMC Pediatrics (2017) 17:169 Page of Fig a Polysomnographic recording during sleep The patient presented suddeneye opening followed by breath-holding with or without agitation and nausea; b Full-montage EEG showingsharp wave discharges intermixed with rhythmic 6–7 Hz activities in the frontal polar and anterior midlineregions during the nocturnal events about 15 s each During the episodes, Sp02 decreased to 87% No epileptiform discharges were noted Gastroscopy results were normal; further, the patient experienced a worsening of her nocturnal symptoms during empirical anti-reflux medication The patient was finally diagnosed with parasomnia and was prescribed clonazepam All symptoms resolved completely within 10 days Seo et al BMC Pediatrics (2017) 17:169 Page of Fig a Polysomnographic recording during nocturnal events The patient was in N3 sleep and presented apnea after arousal; b Results of 24-h pH monitoring showing nocturnal events (black arrows) with low pH (